Recognition of Prodromal Hypoglossal Nerve Palsy Presenting with Neck Pain as Primary Complaint: Findings from a Rare Case Report in Direct Access Physiotherapy during the COVID-19 Pandemic

Neck pain (NP) is the second most common musculoskeletal disorder. Spinal cysts (SCs) are cystic dilatations of the synovial sheaths in joints and tendons. SCs are extremely rare in the cervical spine. Typically, patients are unaware of having an SC due to its asymptomatic nature; however, when cervical SC extends, its volume could compress the surrounding structures, such as the hypoglossal nerve. Isolated hypoglossal nerve palsy (HNP) is very rare and typically presents with unilateral atrophy of the musculature of the tongue and contralateral tongue deviation. Often, patients with HNP also report occipital/neck pain. A 75-year-old man with occipital/neck pain as a primary complaint. Although difficult to observe because of the filtering facepiece two mask, difficulties in articulation and sialorrhea during the interview were noticed. These latter were cues to consider CN examination that revealed CN XII palsy. This prompted a referral for further examination that revealed an SC compressing the right hypoglossal canal. The patient was not considered a surgical candidate and was managed conservatively. This case report outlines the relevant findings relating to the triage of a rare isolated hypoglossal nerve palsy from the physiotherapist’s perspective within a complex setting because of the COVID-19 pandemic. Although referred with a diagnosis of cervical radiculopathy, our case highlights that skilled physiotherapists may play a fundamental role in both the recognition and, when applicable, subsequent novel management of a non-musculoskeletal presentation.


Introduction
Neck pain (NP) is the second most common musculoskeletal disorder being the fourth leading cause of disability, with an annual prevalence rate exceeding 30% [1,2]. In 2017, the Global Burden of Disease reported that both the incidence and prevalence of NP are increasing with age and are greater among females [2]. The physical, psychological, and socioeconomic impact of NP is relevant [1], representing 25% of the workload of outpatient physiotherapy [3]. Additionally, general practitioners are consulted seven times per week for NP [4]. Notably, the number of people who present for a physiotherapy consultation

Case Presentation
A 75-year-old male attended our physiotherapy clinic seeking help for occipital/neck pain. The symptoms started one year prior to the physiotherapy consultation, and at the time, were intermittent, with a 5/10 NPRS resting pain. The pain was worst during the morning and at night-time when lying in bed for sleeping. He was used to doing Pilates and swimming regularly. However, the subject was forced to dismiss any activity because of the pandemic restrictions, having a progressive worsening in the intensity and frequency of his neck pain. Recently, the subject had a left total hip arthroplasty well recovered after a short period of rehabilitation. After surgery, he noticed a progressive worsening of his NP, having his worst pain with 7/10 NPRS and noticeable difficulties in active cervical rotational movements. He also noticed a new spontaneous intermittent sharp pain just below the base of the skull in the upper cervical spine, most noticeable with right rotational movements, which were the most restricted movements. He reported a recent onset-2 months ago-of paresthesia on the fifth finger of his right hand, progressively changing into markable hypo-aesthesia. His general practitioner suggested radiographic scans without physically visiting him as a preauction for the COVID-19 pandemic (i.e., telehealth). Imaging revealed a synostosis at C4-5, a retrolisthesis of C6-7, and an anterolisthesis of C7-T1 ( Figure 1. Note that radiography was not available during the physiotherapy consultations, and the physiotherapist did not feel relevant to having a view after the visit, as described in the following paragraphs. However, for clarity and transparent reporting, the authors felt it relevant to report the finding with an MRI image); thus, the general practitioner suggested physiotherapy and some painkiller for suspected cervical radiculopathy in a follow-up consultation. The subject did report familiarity with rheumatoid arthritis by his mother's family side (i.e., aunt and mother). No other red flags, trauma, head/neck injury, or significant concomitant pathologies were reported. The main reasons for the patient to seek physiotherapy care were to receive explanations of his symptoms, to receive treatment for pain reduction, to reduce movement deficits, and advice for neck exercises and self-management strategies.
signed following the CARE guidelines [40].

Case Presentation
A 75-year-old male attended our physiotherapy clinic seeking help for oc pain. The symptoms started one year prior to the physiotherapy consultation time, were intermittent, with a 5/10 NPRS resting pain. The pain was worst morning and at night-time when lying in bed for sleeping. He was used to do and swimming regularly. However, the subject was forced to dismiss any activ of the pandemic restrictions, having a progressive worsening in the intensi quency of his neck pain. Recently, the subject had a left total hip arthroplasty ered after a short period of rehabilitation. After surgery, he noticed a progress ing of his NP, having his worst pain with 7/10 NPRS and noticeable difficulti cervical rotational movements. He also noticed a new spontaneous intermi pain just below the base of the skull in the upper cervical spine, most noticeabl rotational movements, which were the most restricted movements. He report onset-2 months ago-of paresthesia on the fifth finger of his right hand, pr changing into markable hypo-aesthesia. His general practitioner suggested ra scans without physically visiting him as a preauction for the COVID-19 pan telehealth). Imaging revealed a synostosis at C4-5, a retrolisthesis of C6-7, an rolisthesis of C7-T1 ( Figure 1. Note that radiography was not available during otherapy consultations, and the physiotherapist did not feel relevant to hav after the visit, as described in the following paragraphs. However, for clarity parent reporting, the authors felt it relevant to report the finding with an M thus, the general practitioner suggested physiotherapy and some painkiller fo cervical radiculopathy in a follow-up consultation. The subject did report fami rheumatoid arthritis by his mother's family side (i.e., aunt and mother). No oth trauma, head/neck injury, or significant concomitant pathologies were reported reasons for the patient to seek physiotherapy care were to receive explana symptoms, to receive treatment for pain reduction, to reduce movement defic vice for neck exercises and self-management strategies. .

Physical Examination
A revision of the systems was performed with the goal of excluding any specific pathologies underlying the clinical presentation. His wrist, hand, and finger presented restriction during passive mobilization bilaterally. Although rheumatoid arthritis was never diagnosed, he presented symmetrical finger swelling and deformation on both hands. Additionally, he presented atrophy on his right thenar eminence. Accordingly, a peripheral neurological examination was performed: pinprick, cold, and light touch sensibility were reduced on the ulnar territory of his right hand; in addition, a reduction in force for the first digit adduction and finger extension was observed. Upper limb neurodynamic test (ULNT) 1 testing was unremarkable because of the extension mobility reduction of both wrists. Palpation of the nerve root at the scalene muscle reproduced pain on the neck and shoulder, and by the time his familiar paresthesia. His neck's active right rotation was 50 degrees, with a sharp pain at the base of the skull at the end of the permitted right rotation. Although difficult to observe because of the FFP2 mask, difficulties in articulation and sialorrhea during the interview were noticed. These latter were cues to consider CNs examination: particularly, CN XII testing revealed atrophy and asymmetrical deviation of the tongue toward the affected side (i.e., the right) ( Figure 2 and Video S1). Drooling was also observed (i.e., decreased clearance of saliva) and power reduction of the tongue when tested against the physiotherapist's resistance outside the check while the patient was pushing out his cheek with the tongue (Video S2). No further examination was performed. We recommend readers integrate this case with the article of Taylor et al. [7] for further details on a function-based approach to cranial nerve testing and Mourad et al. [9] for a screening for referral decision tool.

Physical Examination
A revision of the systems was performed with the goal of excluding any thologies underlying the clinical presentation. His wrist, hand, and finger pr striction during passive mobilization bilaterally. Although rheumatoid arthriti diagnosed, he presented symmetrical finger swelling and deformation on both ditionally, he presented atrophy on his right thenar eminence. Accordingly, a neurological examination was performed: pinprick, cold, and light touch sens reduced on the ulnar territory of his right hand; in addition, a reduction in f first digit adduction and finger extension was observed. Upper limb neurod (ULNT) 1 testing was unremarkable because of the extension mobility reduc wrists. Palpation of the nerve root at the scalene muscle reproduced pain on th shoulder, and by the time his familiar paresthesia. His neck's active right rota degrees, with a sharp pain at the base of the skull at the end of the permitted rig Although difficult to observe because of the FFP2 mask, difficulties in artic sialorrhea during the interview were noticed. These latter were cues to consid amination: particularly, CN XII testing revealed atrophy and asymmetrical d the tongue toward the affected side (i.e., the right) ( Figure 2 and Video 1). Dr also observed (i.e., decreased clearance of saliva) and power reduction of the to tested against the physiotherapist's resistance outside the check while the p pushing out his cheek with the tongue (Video 2). No further examination was We recommend readers integrate this case with the article of Taylor et al. [7] details on a function-based approach to cranial nerve testing and Mourad et screening for referral decision tool.  Video S1. CN XII testing revealed asymmetrical deviation of the tongue toward the affected side (i.e., right side).
Video S2. CN XII testing revealed power reduction of the tongue when tested against the physiotherapist's resistance outside the check while the patient was pushing out his cheek with the tongue. Notice the power reduction toward the opposite side (i.e., the left side) and the increased force needed by the physiotherapist to resist when assessing the deviation toward the right. The genioglossus muscles receive motor innervation by the ipsilateral hypoglossal nerve, and its contraction provides tongue deviation toward the opposite side. Lower motor neuron palsy (i.e., at the level or below the level of the nucleus) of the hypoglossal nerve determines contralateral deviation power reduction of the tongue, resulting in a tongue movement toward the palsy side. Notice also the need for the patient to remove the excess saliva in the mouth beyond the lip at the end of the test.

Analysis
Because the patient's presentation was suggestive of CN palsy, the decision was made that he was in need of a referral to a neurosurgeon. Although the patient was wearing an FFP2 mask due to the COVID-19 pandemic, and even if the patient did not think to mention it spontaneously during the history taking, difficulties in articulation and sialorrhea during the interview were noticed. After noticing it, the patient reported having also experienced also recent speech (i.e., articulation) difficulties and drooling, which were a cue to consider testing the CNs, revealing a hypoglossal nerve palsy. That is, it is critical that clinicians ask questions for the recognition of transient or subtle antecedent neurological signs and symptoms and to be able to interpret the complex presentations that commonly combine neck pain with associated disorders [5][6][7][8]18,41]. The process of interpreting the data from the patient history and defining the patient's risk profile is essential to an effective evaluation, including the physical examination: CN examination should be an integral part of that process, having the objective of triaging those patients in need for further examination or referral [5][6][7][8]18,41]. In our case, the CN examination allowed the physiotherapists to offer a much clearer referral, making a recommendation for further examination and which offered both a reported suspicion of compressive palsy and an anatomical clue to the potential pathology [5][6][7][8]18,41]. That is, the sound knowledge of the dysfunction and the recognition of pathological patterns allowed the clinician to dispositionally weigh subtle cues gained in a complex and confounding scenario (i.e., the FFP2 mask due to the COVID pandemic situation and the unreported neurological signs) and that lead to a targeted physical examination [5][6][7][8]18,41].
The hypoglossal nerve is a purely motor nerve, and its composition is very complex. It is the 12th cranial nerve, and it's the most medial of the lower cranial nerves [17]. The hypoglossal nerve is divided into five segments: the first or medullary segment, the second or cisternal segment, the third or skull base segment, the fourth or nasopharyngeal carotid segment, and the fifth or sublingual segment. It arises from the hypoglossal nucleus in the medulla and emerges as multiple radicles between the pyramid and olive [42]. The nerve passes anterolaterally into the posterior cranial fossa and through the hypoglossal canal into the cranium. The occipital condyles (in their inferior portion), the jugular foramen, the jugular process of the occipital bone, and the sphenoid part of the clivus comprise the hypoglossal canal [17,42]. After passing through the canal, the hypoglossal nerve exits the neurocranium. Then, it passes inferiorly in the neck, under the sternocleidomastoid muscle, over the internal carotid artery, and medially to the 9th, 10th, and 11th cranial nerves. The nerve travels deep to the posterior belly of the digastric muscle and crosses the lateral surface of the external carotid artery, reaching the anterior border of the sternocleidomastoid [42]. The hypoglossal nerve usually travels inferiorly to the Wharton duct and lingual artery and ends by innervating the tongue musculature, particularly the extrinsic and intrinsic muscles of the tongue, except the palatoglossus muscle, which is supplied by the vagus nerve. At this level, the nerve joins general somatic efferent fibers from the first cervical nerve C1 [42]. The latter courses with the hypoglossal nerve for a short distance and then drifts away to innervate the geniohyoid, the thyrohyoid muscles, as well as the infrahyoid muscles. The hypoglossal nerve, throughout its course, receives fibers from the nodose ganglion of the 10th cranial nerve at the level of the hypoglossal canal and postganglionic sympathetic fibers from the superior cervical ganglion at the level of the atlas [42]. It also communicates with the lingual branch of the mandibular nerve at the base of the tongue. The sternocleidomastoid muscle can be additionally innervated from the C1 nerve traveling in conjunction with the hypoglossal nerve [42]. Additionally, a combined articular branch derived from a plexus formed by the hypoglossal and C1 nerves entering the atlanto-occipital joint on its lateral surface just superior to the transverse process of the atlas. The hypoglossal nerve regulates all tongue activity and its movement [42].

Follow-Up and Outcomes
The neurosurgeon suggested a magnetic resonance imaging (MRI) scan with contrast to the craniocervical region. The MRI revealed a 9 × 12 × 15 mm hypointense mass in the posterior cranial fossa on T1-weighted images; the T2-weighted images detailed a hyperintense synovial cyst (SC) adjacent to the right side of the craniocervical joint, that was compressing the right hypoglossal canal (Figure 3).
Healthcare 2022, 10, 6 of 13 nerve at the base of the tongue. The sternocleidomastoid muscle can be additionally innervated from the C1 nerve traveling in conjunction with the hypoglossal nerve [42]. Additionally, a combined articular branch derived from a plexus formed by the hypoglossal and C1 nerves entering the atlanto-occipital joint on its lateral surface just superior to the transverse process of the atlas. The hypoglossal nerve regulates all tongue activity and its movement [42].

Follow-Up and Outcomes
The neurosurgeon suggested a magnetic resonance imaging (MRI) scan with contrast to the craniocervical region. The MRI revealed a 9 × 12 × 15 mm hypointense mass in the posterior cranial fossa on T1-weighted images; the T2-weighted images detailed a hyperintense synovial cyst (SC) adjacent to the right side of the craniocervical joint, that was compressing the right hypoglossal canal (Figure 3).
. The delayed diagnosis of SC may have been due to the inadequate evaluation of imaging (i.e., radiography), not only inadequate examination due to telehealth, as MRI evaluation, including T2WI modality, is warranted to detect SC. The neurosurgeon felt that the patient was not a surgical candidate for the SC removal (i.e., risk-benefit ratio), and a cautious physiotherapy program was advised for the patient's cervical and radicular symptoms. For a detailed description of the timeline management, see Figure 4. The delayed diagnosis of SC may have been due to the inadequate evaluation of imaging (i.e., radiography), not only inadequate examination due to telehealth, as MRI evaluation, including T2WI modality, is warranted to detect SC. The neurosurgeon felt that the patient was not a surgical candidate for the SC removal (i.e., risk-benefit ratio), and a cautious physiotherapy program was advised for the patient's cervical and radicular symptoms. For a detailed description of the timeline management, see Figure 4.  The patient was treated one time per week for three months with a multimod gram consisting of gentle manual therapy to the neck (i.e., traction, mobilization, an bilization with movement), neurodynamic to the affected upper limb (ULNT 1 and ers and tensioner), electric dry needling to the upper cervical muscles and the neural of the affected upper limb, with the goal to modulate symptoms. Mobility and str ening exercises to the cervical and scapulothoracic region with progressive load we scribed; also, specific exercises for the recovery of the function of the oro-facialmuscles, of the articulation, and of the tongue force were also delivered. At the follo the subject's pain resolved over time with an improvement of his right neck rotatio 70 degrees), and his peripheral neurological symptoms got better; however, althou speech and sialorrhea improved over time, he continued to present deviation and reduction of the tongue. The capability of the physiotherapist to appropriately re patient and the gentle progressive management strategies were a strength to the peutic alliance, leading the patient to fully adhere to and tolerate the treatment pla hough the aim of the present case is to describe the physiotherapist's screening f referral process, we invite readers to refer to Table 1 for more details related to the ments. Week 39 Week 40 Week 40 Week 40 Week 41 Week 41 Week 41 Week 52 The patient was treated one time per week for three months with a multimodal program consisting of gentle manual therapy to the neck (i.e., traction, mobilization, and mobilization with movement), neurodynamic to the affected upper limb (ULNT 1 and 3 sliders and tensioner), electric dry needling to the upper cervical muscles and the neural tissue of the affected upper limb, with the goal to modulate symptoms. Mobility and strengthening exercises to the cervical and scapulothoracic region with progressive load were prescribed; also, specific exercises for the recovery of the function of the oro-facialbuccal muscles, of the articulation, and of the tongue force were also delivered. At the follow-up, the subject's pain resolved over time with an improvement of his right neck rotation (i.e., 70 degrees), and his peripheral neurological symptoms got better; however, although his speech and sialorrhea improved over time, he continued to present deviation and power reduction of the tongue. The capability of the physiotherapist to appropriately refer the patient and the gentle progressive management strategies were a strength to the therapeutic alliance, leading the patient to fully adhere to and tolerate the treatment plan. Although the aim of the present case is to describe the physiotherapist's screening for the referral process, we invite readers to refer to Table 1 for more details related to the treatments.

Specific exercise therapy
To improve oro-facial-buccal functions, including the tongue Frequent daily home exercise program focused on tongue function (e.g., isometric strengthening exercises of the tongue, especially in abduction, and articulation exercises to lips, tongue, and mouth to improve physical function) Very low tongue abduction power (i.e., not able to overcome small resistance) measured by the physiotherapy hand resistance Subjective perception of the amount of drooling measured by the frequency of lips clearance = "very frequent" Moderate tongue abduction power (i.e., able to overcome a small/moderate resistance isometrically) measured by the physiotherapy hand resistance Subjective perception of the amount of drooling measured by the frequency of lips clearance = "50% less frequent"

Discussion
Red flags are signs and symptoms that should alert physiotherapists to consider carefully if the patient is within the scope of practice or whether an appropriate medical referral is needed [5][6][7][8][9]43,44]. Commonly, patients with serious pathology of the neck present subtle transient antecedent neurological signs or symptoms (e.g., acute onset of unusual headache or NP, and/or ischemic signs and symptoms) or risk factor (e.g., recent head/neck trauma) [5][6][7][8][9]43,44]. An increasing body of evidence suggests neurological examination (i.e., peripheral nerves, CNs, and upper motor neuron testing) as an important competence for physiotherapists when examining for potentially serious conditions of the neck [5][6][7][8][9]43,44]. Typically, due to their anatomical location, the inferior portion of CNs (i.e., CN V trigeminal, CN VII facial, CN IX glossopharyngeal, and CN X vagus nerves, CN XII hypoglossal) and the nerves of the superior cervical spine (e.g., the occipital nerve) are involved in facial syndromes and NP [9]. That is, the peripheral extraforaminal segments of these nerves travel below the base of the skull [17]; thus, their involvement should be considered in cases of upper neck pain/headache, neuralgic pain, disturbed speech, swallowing, coughing, deglutition, sensory dysfunctions, taste, autonomic dysfunctions, dysphagia, pharyngeal pain, cardiac or gastrointestinal compromise, and weakness of the trapezius, of the sternocleidomastoid, or of the tongue muscles [7,42].
In our case, the NP associated with the onset of hypo-aesthesia on the fifth finger of the patient's right hand were the main symptoms that led the medical doctor to refer the subject for physiotherapy. The complaint of a new spontaneous intermittent sharp pain just below the base of the skull, the rapid increase in the intensity of neck pain, the marked reduction in right rotational ROM, the difficulties in speaking, and the sialorrhea led the dispositional clinical reasoning of the physiotherapist to the decision that the patient's condition required a referral to a neurosurgeon [5,7,14,15]. The following MRI investigation detected an atlantooccipital SC, which was compressing the hypoglossal nerve along its canal, causing an isolated HNP. MRI with or without contrast of the skull base and brain focusing the hypoglossal nerve and its course, and MRI neurography, an evolution of conventional MRI, should be used while evaluating the cause of suspected HNP [17,28]. In addition, high-resolution basicranial computerized tomography (CT) scans may be considered when evaluating osseous involvement, such as degenerative osteoarticular diseases or craniovertebral junction trauma [17,28].
Spinal SC is extremely rare in the cervical spine and manifests during the fifth decade with no gender predominance [45]. SC are cystic dilatations of the synovial sheaths that are often found in joints and tendon sheaths. The etiology of spinal SC is unclear, and many theories have been proposed to explain their pathogenesis [45,46]. Possible causes include synovial fluid extrusion from the joint capsule, increased motion at the spinal joint, myxoid degeneration, cyst formation in the synovial tissue, and latent growth of developmental rests of synovial tissue or tissue metaplasia [47]. These cysts, when degenerative, result from an extrusion of the synovium through a capsular because of a degenerated or unstable facet joint [47]. SCs in the neck involve both the atlanto-odontoid process articulation (with or without atlanto-axial dislocation), C1-C2 zygapophyseal articulations, and, more rarely, the atlanto-occipital joints [45]. Typically, patients are unaware of having an SC due to its asymptomatic nature; however, when an SC extends its volume and invades and compresses the surrounding structures, it may cause nerve root or spinal cord compression resulting in pain and/or neurological symptoms [48]. In our case report, a 75-year-old male with an atlantoaxial SC had symptoms related to the isolated compression of the right hypoglossal nerve related to the close anatomical relationship between the hypoglossal nerve and the atlanto-occipital joint.
The management of the SC is still under debate, and several treatments have been suggested, including surgical resection, CT-guided cyst surgical aspiration, or steroid facet injection. However, a conservative approach using collars [49][50][51] or external spine immobilization has been shown to have benefits for cyst regression [45]. Notably, when the SC is located at the cervical level and presents the potential to compress any vital structures, surgery is the treatment of choice [45]; the approach choice depends on the surgeon's preference and on the extent of the SC [47,52]. Lateral suboccipital craniectomy or juxta condylar approaches are the most described surgical procedures. Generally, cysts located at different sites may resolve spontaneously with clinical improvement; however, data on craniocervical SC behavior and spontaneous regression are not available. To the best of our knowledge, only one case report described a spontaneous SC resolution associated with neurological symptom improvement [45].
To the best of the authors' knowledge, this is the first case report that describes the clinical reasoning and the decision-making process that led a physiotherapist to refer a patient with SC causing an isolated HNP presenting neck/occipital pain. However, our article presents a single episode of care and may represent an outlier in clinical practice, necessitating caution regarding generalizability. In addition, a multimodal management approach consisting of manual therapy and specific exercises directed to the tongue, the neck, and shoulders was never described before for the treatment of HNP. The benefits provided by physiotherapy may rely on the neuroanatomical and functional relationship between the cervical spine and the stomatognathic system; that is, alterations in the lips, the tongue, the jaw, the cheeks, and the stomatognathic functions have been already described in the literature as orofacial myofunctional disorders (OMDs) [53]. OMDs can impair both the cervical and the temporomandibular function being reciprocally contributing factors [54]. In our case, HNP secondary to an SC compression at the atlanto-occipital joint compromised the patient's orofacial function, potentially triggering NP by the convergence of cervical nociceptive afferents at the level of the cervical trigeminal nucleus. The afferents fibers from the lingual nerve (trigeminal and hypoglossal anastomosis) traveling to the upper cervical ventral ramus provide a plausible explanation for both neck symptoms and the maladaptive response of the orofacial musculature to the impaired function on the tongue [55,56]. All of the above lead the authors to believe that the present case may represent a novel approach for the management of isolated HNP.

Conclusions
Neck pain is commonly encountered in physiotherapy outpatient settings, and delayed diagnosis of serious presentation masquerading musculoskeletal NP is frequent. With physiotherapists assuming the responsibility of the first contact role, it is essential that physiotherapists possess the triage skills (namely, screening for referral) to recognize the key elements of the patient history and the physical examination which may be indicative of a condition outside the scope of practice. This case report outlines the relevant aspects relating to the pathophysiology, screening, and differential diagnosis of an isolated hypoglossal nerve palsy secondary to degenerative changes of the cervical spine. It also describes the relevant findings from the history and physical examination from the physiotherapist's perspective of an old patient with a rare condition within a complex and confounding scenario. Although the patient was referred by a physician with a diagnosis of cervical radiculopathy, our case highlights that skilled physiotherapists may play a fundamental role in both the recognition and subsequent novel management of a non-musculoskeletal presentation.
Supplementary Materials: The following supporting information can be downloaded at: https:// www.mdpi.com/article/10.3390/healthcare11091342/s1. Video S1. CN XII testing revealed asymmetrical deviation of the tongue toward the affected side (i.e., right side). Video S2. CN XII testing revealed power reduction of the tongue when tested against the physiotherapist's resistance outside the check while the patient was pushing out his cheek with the tongue.

Institutional Review Board Statement:
The study was conducted in accordance with the Declaration of Helsinki. According to local legislation, ethics approval is not required for case reports (Regolamento del "Comitato Etico nazionale per le sperimentazioni degli enti pubblici di ricerca (EPR) e altri enti pubblici a carattere nazionale", presso l'Istituto Superiore di Sanità, Art. 2-Funzioni, 11 April 2022).

Informed Consent Statement:
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal.